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Báo cáo khoa học: Binding of ATP at the active site of human pancreatic glucokinase – nucleotide-induced conformational changes with possible implications for its kinetic cooperativity doc

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Here, we report on a conformational change induced by the binding of adenine nucleotides to human pancreatic GK, as determined by intrinsic tryptophan fluorescence, using the catalyticall

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glucokinase – nucleotide-induced conformational changes with possible implications for its kinetic cooperativity

Janne Molnes1,2,3, Knut Teigen3, Ingvild Aukrust1,2,3, Lise Bjørkhaug2,4, Oddmund Søvik2, Torgeir Flatmark3and Pa˚l Rasmus Njølstad1,2

1 Department of Pediatrics, Haukeland University Hospital, Bergen, Norway

2 Department of Clinical Medicine, University of Bergen, Norway

3 Department of Biomedicine, University of Bergen, Norway

4 Center for Medical Genetics and Molecular Medicine, Haukeland University Hospital, Norway

Introduction

Glucokinase (GK) or hexokinase IV (EC 2.7.1.1)

catal-yses the phosphorylation of a-d-glucose (Glc) to form

glucose 6-phosphate, the entry point of Glc into

gly-colysis, using MgATP2) as the phosphoryl donor

Human GK (hGK) is expressed in the liver [1], pan-creas [2], brain, and endocrine cells of the gut [3,4] It

is a key regulatory enzyme in the human pancreatic b-cell (isoform 1), playing a crucial role in the regulation

Keywords

ATP binding; catalytic mechanism; GCK

maturity onset diabetes of the young

(GCK-MODY); glucokinase; kinetic cooperativity

Correspondence

T Flatmark, Department of Biomedicine,

University of Bergen, N-5009 Bergen,

Norway

Fax: +47 55586360

Tel: +47 55586428

E-mail: torgeir.flatmark@biomed.uib.no

Note

The atomic coordinates of the molecular

dynamics simulated structural models are

available from knut.teigen@biomed.uib.no

(Received 7 April 2011, revised 20 April

2011, accepted 4 May 2011)

doi:10.1111/j.1742-4658.2011.08160.x

Glucokinase (GK) is the central player in glucose-stimulated insulin release from pancreatic b-cells, and catalytic activation by a-D-glucose binding has

a key regulatory function Whereas the mechanism of this activation is well understood, on the basis of crystal structures of human GK, there are no similar structural data on ATP binding to the ligand-free enzyme and how

it affects its conformation Here, we report on a conformational change induced by the binding of adenine nucleotides to human pancreatic GK, as determined by intrinsic tryptophan fluorescence, using the catalytically inactive mutant form T228M to correct for the inner filter effect Adeno-sine-5¢-(b,c-imido)triphosphate and ATP bind to the wild-type enzyme with apparent [L]0.5 (ligand concentration at half-maximal effect) values of 0.27 ± 0.02 mM and 0.78 ± 0.14 mM, respectively The change in protein conformation was further supported by ATP inhibition of the binding of the fluorescent probe 8-anilino-1-naphthalenesulfonate and limited proteol-ysis by trypsin, and by molecular dynamic simulations The simulations provide a first insight into the dynamics of the binary complex with ATP, including motion of the flexible surface⁄ active site loop and partial closure

of the active site cleft In the complex, the adenosine moiety is packed between two a-helices and stabilized by hydrogen bonds (with Thr228, Thr332, and Ser336) and hydrophobic interactions (with Val412 and Leu415) Combined with enzyme kinetic analyses, our data indicate that the ATP-induced changes in protein conformation may have implications for the kinetic cooperativity of the enzyme

Abbreviations

AdN, adenine nucleotide; AMP-PNP, adenosine-5¢-(b,c-imido)triphosphate; ANS, 8-anilinonaphthalene-1-sulfonate; ATPcS, adenosine-5¢-O-(3-thiotriphosphate); GCK-MODY, GCK maturity-onset diabetes of the young; GK, glucokinase; GKA, glucokinase activator; Glc, a- D -glucose; GST, glutathione-S-transferase; hGK, human glucokinase; ITF, intrinsic tryptophan fluorescence; MD, molecular dynamic; n H , Hill coefficient; PDB, Protein Data Bank; WT, wild-type.

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of insulin secretion, and is therefore termed the

pancre-atic b-cell glucose sensor [5] In humans, more than

600 different mutations in the glucokinase gene (GCK)

have been detected in patients suffering from familial,

mild fasting hyperglycaemia [GCK maturity onset

dia-betes of the young (GCK-MODY), GCK permanent

neonatal diabetes mellitus, and GCK congenital

hyper-insulinism of infancy [6–11] Some of the mutations

greatly reduce the binding affinity of MgATP2)

[11,12], which is compatible with a direct interaction of

these residues with the nucleotide at the active site

The catalytic mechanism of GK has been the subject

of several detailed analyses, and is still a partly

unre-solved issue Although some theoretical evidence has

been presented in support of a random order

mecha-nism, in which the enzyme interacts with the substrate

and cosubstrate in a random fashion [13], enzyme

kinetic studies support an ordered mechanism in which

Glc binds to the enzyme before the cosubstrate [14–

16] The discussion is reminiscent of that related to the

catalytic mechanism of yeast hexokinase [17] For both

enzymes, part of the discussion has been related to the

question of whether ATP binds to the Glc-free enzyme

and the possibility of a nucleotide-triggered change in

protein conformation

In this work, we have studied the interaction of

ATP and analogues with the human pancreatic enzyme

with the aims of: (a) presenting experimental evidence

for equilibrium binding to the ligand-free super-open

conformation; (b) demonstrating possible

conforma-tional changes associated with ATP binding; (c)

obtaining insights into the active site contact residues

involved in ATP binding; and (d) relating this

informa-tion to steady-state enzyme kinetic data To achieve

these aims, we used a combined experimental approach

including intrinsic tryptophan fluorescence (ITF),

extrinsic 8-anilino-1-naphthalenesulfonate (ANS)

fluo-rescence, limited proteolysis, and molecular dynamic

(MD) simulations Additionally, enzyme kinetic

analy-ses were performed to evaluate the functional

implica-tions of the structural data The different approaches

provide new insights into the interaction of ATP with

hGK, with possible implications for the positive kinetic

cooperativity with respect to Glc

Results

Recombinant proteins

The average yields of soluble recombinant pancreatic

glutathione-S-transferase (GST)–hGK fusion proteins

were  4.0 mg L)1 (wild type and T228M) and

 2.0 mg L)1 (L146R) As the recombinant wild-type

(WT) hGK and WT GST–hGK enzymes demonstrate similar steady-state kinetic parameters and the same apparent Kd for Glc in the ITF equilibrium binding assay [18], the fusion proteins were used in kinetic studies and ITF equilibrium binding analyses with Glc

In the adenine nucleotide (AdN) equilibrium binding studies, we compared nontagged and GST-tagged GK

In all other experiments, only the nontagged proteins were used

Characterization of the T228M mutant reference enzyme

The T228M mutant form, causing GCK-MODY in the heterozygous state and GCK permanent neonatal dia-betes mellitus in the homozygous state [9,19], was selected as a non-ATP-binding reference enzyme on the basis of its previously described kinetic properties [9,20,21] Here, equilibrium binding of Glc, as deter-mined by ITF, demonstrated an increased affinity (Kd= 3.1 ± 0.1 mm) in comparison with WT GST– hGK (Kd= 4.3 ± 0.1 mm), and a fluorescence enhancement signal response [(DFeq⁄ F0)max· 100] simi-lar to that of the wild type (Table 1) Steady-state kinetic analyses demonstrated a  9000-fold reduced catalytic activity (kcat 7 · 10)3s)1) (Table 1) Thr228 is a highly conserved residue at the active site

of the hexokinase family of enzymes, positioned in the phosphate-binding loop and part of a classical ATP-binding motif (phosphate 2 site) in hexokinases and homologous proteins [22] In the crystal structures of

Table 1 The steady-state kinetics and ITF properties of WT GST– hGK and two GCK-MODY mutant forms NM, not measurable.

kcat(s)1) c 67.6 ± 1.3 7 · 10)3 0.77 ± 0.03

Relative catalytic activity (%)

KmMgATP2)(m M ) 0.16 ± 0.01 NM 0.24 ± 0.04 Hill coefficient (nH) c 1.95 ± 0.19 NM 1.29 ± 0.04 Hill coefficient (n H ) d 1.15 ± 0.04 NM 0.73 ± 0.04 Glc response (%)

[(DFeq⁄ F o )max· 100]

28.7 ± 1.5 29.2 ± 0.1 5.3 ± 0.5

K d Glc (m M ) e 4.3 ± 0.1 3.1 ± 0.1 19.3 ± 3.8

a The n H , [S] 0.5 and K d values were not measured, because of low catalytic activity. bThe ITF responses to 200 m M Glc were 33.2 and 36.0 arbitrary fluorescence units for the fusion protein and the isolated T228M hGK mutant, respectively c Assay with Glc as the variable substrate. dAssay with ATP as the variable substrate.

e Obtained from equilibrium binding measurements by intrinsic Trp fluorescence spectroscopy.

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human and yeast hexokinases, the hydroxyl group of

this conserved Thr interacts with the a-phosphate of

ATP [21,23,24], and a Thrfi Met substitution in hGK

is inferred to eliminate this important contact (see the

in silico studies below) According to the coordinates

of the closed (Glc-bound) conformation of WT hGK

[Protein Data Bank (PDB) ID 1v4s], the T228M

mutation is predicted to be destabilizing, as measured

by the free energy of thermal unfolding (DDG =

)4.07 kcalÆmol)1) and the free energy of folding

(DDG = 0.85 kcalÆmol)1) However, the far-UV CD

spectrum was very similar, if not identical, to that of

WT hGK (Fig S1), and no significant differences in

the apparent Tmvalues (on thermal unfolding) of WT

hGK (Fig 1) and the mutant protein (data not shown)

were observed Thus, the Thrfi Met substitution has

little impact on the protein fold

Equilibrium binding of adenosine-5¢-(b,c-imido)

triphosphate (AMP-PNP), ATP and MgATP to the

ligand-free enzyme

To study binding of AdNs to the ligand-free nontagged

enzyme, we first measured the change in ITF

[(DFeq⁄ F0)· 100] at 25 C as a function of the AdN

concentration In contrast to the enhancement of the

ITF signal observed with Glc [18,25], the ATP analogue

AMP-PNP resulted in quenching of the fluorescence

(Fig 2), consistent with a previous report [26] However,

the inner filter effect resulting from nucleotide

absor-bance at the excitation wavelength (295 nm), which was

not considered in that report, made a significant

contri-bution to the quenching To correct for this effect, a

sim-ilar titration was performed with the non-ATP-binding

mutant T228M and with free Trp (Fig 2A,C) Of the

two reference titrations, the T228M mutant gave the

preferred correction (Fig 2A), as the mutant also

dem-onstrated quenching of the ITF at low concentrations

(£ 0.1 mm) From the fluorescence difference data

(Fig 2B), an apparent [L]0.5 (ligand concentration at

half-maximal effect) value of 0.27 ± 0.02 mm (25C)

was estimated by nonlinear regression analysis The net

(specific) fluorescence quenching observed for AMP-PNP

was modest, but significant [D(DFeq⁄ F0)max· 100 =

)2.6% ± 0.2%], suggesting that one or more of the

three Trp residues (Trp99, Trp167, and Trp257) undergo

small changes in quantum yield, but without any

signifi-cant spectral shift A similar result was obtained with

the respective GST–hGK fusion proteins (Fig 2C,D),

with an [L]0.5value of 0.16 ± 0.04 mm and D(DFeq⁄ F0

)-max· 100 =)2.2% ± 0.2% In the ITF titrations of

the wild type and the T228M mutant (control) with

increasing concentrations of ATP (Fig 2E), a net

decrease in fluorescence intensity similar to the AMP-PNP response was observed The differential binding data (Fig 2E) were fitted to a hyperbolic binding iso-therm by nonlinear regression (r2> 0.97), giving a half-maximal effect ([L]0.5) at 0.78± 0.14 mm and D(DFeq⁄ F0)max · 100 =)1.5% ± 0.1% Similar titrations with MgATP gave comparable maximal quenching of ITF of D(DFeq⁄ F0)max· 100= )2.2% ± 0.3%

A

B

Fig 1 Thermal refolding–unfolding and aggregation of WT hGK The experiments were performed as described in Experimental pro-cedures (A) The thermal refolding–unfolding profile of WT hGK (23 l M ) in the absence of Glc was determined by following the change in ellipticity at 222 nm at a constant heating rate of

40 CÆh)1 An apparent transition temperature (T m ) of 42.4 ± 0.2 C was determined from the first derivative of the smoothed denatur-ation curve No significant difference in the profile was observed in the presence of Glc (data not shown) The observed optical activity

is expressed as the mean residue molar ellipticity ([h]MR) (B) The pseudo-absorbance data were obtained at the same time as the

CD data in (A), reporting on the biphasic heat-induced increase in absorbance The regression lines, based on data points in the tem-perature interval 24–79 C, indicate an inflection point at  42 C and increasing aggregation of the protein above this temperature; above  80 C, the absorbance decreased, probably owing to pre-cipitation of the protein.

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Thermal refolding and unfolding

As previously demonstrated by ITF, ligand-free WT

hGK senses temperature shifts from 4 to 39C directly

by a slow (seconds to minutes) conformational change

(hysteresis), with a biphasic time course in temperature

jump (4–39C) experiments [18] The far-UV CD

spec-troscopy at 222 nm confirmed this conformational

change by an apparent change in the secondary

struc-ture in the same temperastruc-ture range (Fig 1A) At

higher temperatures, the enzyme demonstrated

rela-tively low global thermodynamic stability, with an

apparent Tmof 42.4 ± 0.2C and increasing

aggrega-tion at temperatures ‡ 42 C, as measured from the

associated high-voltage (pseudo-absorbance) curve

obtained at the same time (Fig 1B) Similarly, the iso-thermal (25C) chemical unfolding caused by guani-dine chloride also resulted in aggregation of the protein (data not shown) This instability of the pro-tein precluded an estimate of equilibrium thermody-namic parameters, and thus also measurement of the effect of ligands on such conformational equilibria

Effect of ATP and Glc on extrinsic ANS fluorescence and limited proteolysis ANS is an extrinsic fluorophore with affinity for hydro-phobic clusters in proteins that are not tightly packed

in a fully folded structure or become exposed in par-tially unfolded structures [27] The weak fluorescence of

[AMP-PNP] (m M )

WT hGK T228M hGK Tryptophan

A

0.0 0.5 1.0 1.5 2.0 2.5 3.0

) ] x 100

[AMP-PNP] (m M )

B

1.0 2.0 3.0 4.0 5.0

[AMP-PNP] (m M )

WT GST-hGK T228M GST-hGK Tryptophan

GST–hGK

C

[AMP-PNP] (m M )

) ] x 100

0.2

0.4

0.6

0.8

1.0

1.2

1.4

1.6

1.8

D

) ] x 100

0.2 0.4 0.6 0.8 1.0 1.2 1.4 1.6

[ATP] (m M )

E

Fig 2 Equilibrium binding of AMP-PNP (A–D) and ATP (E) in the absence of Glc (A) The change in fluorescence intensity [(DFeq⁄ F 0 ) · 100] was measured at 25 C upon subsequent additions of ligand (A) AMP-PNP titration curves of WT hGK (d), the non-ATP-binding mutant T228M hGK (s), and free Trp (at a concentration giving the same F 0 value as the enzyme) (.) The data were fitted to binding isotherms by nonlinear regression analysis, with r 2 > 0.99 for both WT hGK and T228M hGK Data points and error bars represent the mean ± SD of three independent titrations (B) The net fluorescence quenching [D(DFeq⁄ F 0 )max· 100] of WT hGK as a function of [AMP-PNP], with a calculated [L] 0.5 value of 0.27 ± 0.02 m M The data points and the solid line represent the difference between the WT and T228M hGK titrations (C) The same experiment as in (A), but performed on the GST fusion proteins The titration curves of WT GST-hGK (d), the non-ATP-binding mutant T228M GST–hGK (s), and free Trp (at a concentration giving the same F0value as the enzyme) (.) The data were fitted to binding isotherms by nonlinear regression analysis, with r 2 > 0.99 for both WT GST–hGK and T228M GST–hGK (D) The net fluorescence quenching [D(DF eq ⁄ F 0 ) max · 100] of WT GST–hGK as a function of [AMP-PNP], with a calculated [L] 0.5 value of 0.16 ± 0.04 m M The data points and the solid line represent the difference between the WT and T228M GST–hGK titrations (E) Equilibrium binding of ATP to WT GST–hGK in the absence of Glc The figure shows the net decrease in ITF [D(DF eq ⁄ F 0 ) max · 100] with increasing concentrations of ATP (25 C), calculated

in a similar manner as in (B) and (D), representing the difference between the WT GST–hGK and T228M GST–hGK titrations The data were fitted to a hyperbolic binding isotherm by nonlinear regression analysis (r 2 > 0.97), and an [L]0.5value for ATP of 0.78 ± 0.14 m M was calcu-lated The data points (d) represent the means of duplicate titration experiments.

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ANS was greatly enhanced upon binding to ligand-free

WT hGK (Fig 3A), with a maximum at  480 nm

(blue shift), indicative of ANS binding to exposed

hydrophobic clusters As seen from Fig 3B, both ATP

and Glc significantly reduced the ANS fluorescence

sig-nal [Glc (P = 0.00004) > ATP (P = 0.004)],

compati-ble with a decrease in accessicompati-ble hydrophobic clusters

as compared with the ligand-free enzyme

In our studies on mutant forms of hGK, their

sus-ceptibilities to limited proteolysis by trypsin have

proved to be a valuable conformational probe

(unpub-lished data) Here, it was demonstrated (Fig 3C) that

the ligand-free WT hGK (at 25C) is partly stabilized

by its association with ATP and Glc (Glc > ATP)

Effect of nonhydrolysable ATP analogues on the

equilibrium binding of Glc

The equilibrium binding of Glc to the ligand-free WT

hGK and its binary AdN complexes was determined

by its enhancement of the ITF signal (Table 2) In the

absence of AdNs, a hyperbolic binding isotherm for

Glc was observed, with a Kdvalue of 4.2 ± 0.1 mm at

25C Titration with Glc in the presence of

Mg-adeno-sine-5¢-O-(3-thiotriphosphate) (ATPcS) and

MgAMP-PNP also gave hyperbolic binding isotherms; however,

the apparent affinity for Glc increased (Table 2), i.e

about two-fold with 5 mm MgAMP-PNP (P = 0.002)

A similar effect was observed for the GCK-MODY

L146R mutant in the presence of 2.5 mm ATPcS; that

is, the apparent Kd decreased from 19.3 ± 3.8 mm to

14.0 ± 1.4 mm (Fig 4), and there was a 25%

incre-ase in the fluorescence signal response [(DFeq⁄ F0)max·

100] The mutant demonstrated a 100-fold reduction

in kcatand a  40-fold increase in the [S]0.5(substrate

concentration at half-maximal activity) value for Glc

(Table 1) The positive kinetic cooperativity with

respect to Glc was partly lost in the mutant

(nH= 1.29 ± 0.04), and in contrast to previous

find-ings [28], the Km for ATP (0.24 ± 0.04 mm) was only

slightly increased

In silico dynamic and conformational effects of

ATP binding

In the MD simulations, the starting crystal structure

(PDB ID 1v4t) of the ligand-free super-open

confor-mation was modified to include the 23 missing residues

(Glu157–Asn179) in a surface loop structure (see

Experimental procedures) The Ca rmsd value for the

modelled structure and the crystal structure was

 2.3 A˚ when the Glu157–Asn179 loop residues were

not included From the computed B-factor values (Figs

A

B

C

Fig 3 ANS fluorescence measurements and limited proteolysis (A) Emission fluorescence spectra (k ex = 385 nm) of free ANS in buffer and ANS in the presence of 0.75 l M WT hGK A final ANS concentration of 60 l M was used (B) The effect of ATP and Glc on ANS binding to WT hGK The ANS binding experiments were per-formed at a temperature of 38 C, as described in Experimental procedures, with 60 l M ANS and a protein concentration of 0.75 l M The concentrations of Glc and ATP were 30 m M and

2 m M , respectively Each column represents the mean ± SD of three independent experiments Statistical significance was deter-mined with Student’s t-test: **P < 0.01 and ***P < 0.0001 (C) Time-course for the limited proteolysis of WT hGK by trypsin WT GST–hGK (0.5 mgÆmL)1) was cleaved with factor Xa for 2 h at 4 C, and subsequently subjected to limited proteolysis by trypsin at

25 C (trypsin ⁄ hGK ratio of 1 : 400 by mass) in the absence of ligand (d), or in the presence of either 40 m M Glc ( ) or 2 m M ATP ⁄ 4 m M MgAc (s) Data points and error bars represent the mean ± SD of three independent experiments.

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5A and S2B), the region that fluctuates the most is

Glu157–Asn179, consistent with the observed disorder

in the crystal structure MD simulations of the

mod-elled binary GK–ATP complex revealed that the global

rmsd of the structure converged at the end of the 2-ns

simulation period (Fig S2A) The dynamic changes in

the active site cleft opening over the 2-ns equilibration

period (Fig 5C), as defined by the residues Lys169–

Gly223 (‘hinge’)–Gly229, suggest partial closure of the

interdomain cleft ( 15) These defining residues were

previously used to monitor the opening of the cleft

Table 2 The effect of ATP analogues on the equilibrium binding

affinity of Glc as determined by ITF fluorescence titrations on WT

GST–hGK.

MgAMP-PNP

MgATPcS

a Mean ± SD of five independent titration experiments b Based on

nonlinear regression analysis of single binding isotherms (r2> 0.99)

(n = 12 data points) c Mean ± SD of three independent titration

experiments.

Fig 4 The Glc binding isotherm for the mutant L146R GST–hGK.

The enhancement of ITF was measured at 25 C with increasing

concentrations of Glc in the absence (d) and presence (s) of

2.5 m M ATPcS The solid lines represent the fit of the data to two

hyperbolas as obtained by nonlinear regression analyses, giving Kd

values of 19.3 ± 3.8 m M (r 2 > 0.98) and 14.0 ± 1.4 m M (r 2 > 0.99)

in the absence and presence of ATPcS, respectively, and a

fluores-cence signal response [(DF eq ⁄ F 0 ) max · 100] of  5% For

compari-son, the (DFeq⁄ F 0 )max· 100 was  30% for WT GST–hGK Data

points and error bars represent the mean ± SD of three

indepen-dent experiments.

asl

asl

*

70

30 40 50 60

Model 1 Model 3

Time (ps)

10 20

Model 4

A

B

C

Fig 5 (A, B) Computed B-factor values and changes in the interdo-main cleft angle The computed B-factor values for the MD simu-lated model structures of the apoenzyme and the hGK–ATP binary complex The values are colour-coded onto the 3D ribbon structure

of (A) the apoenzyme and (B) the hGK–ATP binary complex, with red corresponding to the most mobile region (B-factor ‡ 400 A˚ 2

), blue corresponding to the most stable region (B-factor £ 40 A˚ 2 ), and green corresponding to B-factor values in the range 40–400 A˚2 Note also the change in secondary structure of the flexible active site loop (asl), comprising residues Ser151–Cys181, on binding of ATP (*) The B-factor values versus residue numbers are shown in Fig S2B (C) The changes in the interdomain cleft angle during the 2-ns MD simulations at 300 K The change in the cleft angle was defined by the residues Lys169–Gly223 (‘hinge’)–Gly229, compati-ble with a partial closure of  15 Model 1: hGK super-open con-formation (including coordinates for the Glu157–Asn179 loop) Model 2: hGK super-open conformation with inserted Glc Model 3: hGK super-open conformation with inserted ATP Model 4: hGK ter-nary complex with Glc and ATP.

Trang 7

( 50) on MD simulations of Glc dissociation from

the binary hGK–Glc complex [29] A molecular

motion was further indicated by the dyndom

algo-rithm [30], with the coordinates obtained for the

ligand-free form and the hGK–ATP complex at the

end of the simulations (Figs 6B,C and S3; Table S2),

also indicating partial closure of the cleft ( 33) and

an apparent domain motion, which were less dramatic

than for the Glc-induced conformational transition

(Table S2) In the final structure of the binary complex

(Fig 6A; Table S1), the adenosine moiety is packed

between helices 12 and 15 in the L-domain [29] and

stabilized by hydrogen bonds (with Thr332 and Ser336

in helix 12) and hydrophobic interactions (with Val412

and Leu415 in helix 15)

A conformational change was also indicated by the

MD simulations of the modelled ternary hGK–Glc–

ATP complex In the final structure of the simulations,

the interactions of the adenosine moiety were similar

to those observed in the binary ATP complex, with the

a-phosphate and b-phosphate oxygen atoms forming

hydrogen bonds with Thr228 and Ser411 in the

L-domain (data not shown)

For comparison, when the MD simulations were

performed with Glc in the super-open conformation

(Fig 5C), no significant change in the interdomain

cleft was observed The substrate was found to be

positioned at the active site, as expected [18], including

the interactions with the primary contact residues

Asn204 and Asn231 (data not shown) However, no

interactions with Thr168 and Lys169 were seen, as the

Ser151–Val181 surface loop was not displaced in the

direction of Glc, and there was no measurable closure

of the active site cleft during the 2-ns MD simulations

(Fig 5C), as observed in the crystal structures of the

binary GK–Glc complex [18,31] Thus, in this case, the

simulation time (2 ns) was too short to demonstrate

the large global conformational transition observed by

ITF upon Glc binding, which has a millisecond to

minute time scale [18,25,32], characteristic of this

hys-teretic enzyme, and thus out of reach of

nanosecond-scale MD simulations

Steady-state kinetics

The steady-state kinetic properties of WT GST-hGK

were determined with Glc as the variable substrate at

high or low concentrations of MgATP (Table 3)

Posi-tive cooperativity was observed with respect to Glc at

5 mm (saturating) MgATP (nH= 1.95 ± 0.10)

(Fig 7A) with an [S]0.5 value of 8.2 ± 0.3 mm

How-ever, at 0.05 mm MgATP, the cooperativity was reduced

to nH= 1.07 ± 0.07 (Fig 7B), and the [S]0.5value was

A

**

B

C

D205

R447

K169

K296

T332

S336

L415 V412 S411

T228

ATP

[helix 12]

[helix 15]

αα

β γ

Fig 6 The ATP-binding site in the MD simulated model structure

of the binary hGK–ATP complex and the domain motion induced by ATP binding to the hGK apoenzyme (A) Close-up view of the ATP-binding site in the MD simulated model structure of the binary hGK-ATP complex, showing the main contact residues with ATP; for a presentation of all contact residues, see Table S1 For helix nomenclature, see [47] (B, C) The domain motion induced by ATP binding to the apoenzyme with partial closure of the active site cleft and a rotation angle of  33 The coordinates were those obtained for (B) the modelled super-open conformation, including the Glu157–Asn179 loop, and (C) the modelled open conformation with inserted ATP (GK–ATP) The Carmsd values were 4.01 A ˚ for the whole protein, 2.09 A ˚ for the fixed domain (349 residues), and 3.91 A ˚ for the moving domain (87 residues) The dynamic domains were identified with the DYNDOM program [30] The Cabackbone structures, shown in line presentation, were colour-coded as fol-lows: blue, fixed domain; red, moving domain; and green, connect-ing residues For comparison, the correspondconnect-ing data for the domain motion induced by Glc binding to the apoenzyme are shown in Table S2 **ATP.

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increased to 14.3 ± 1.7 mm The fact that the kinetic

cooperativity is dependent on the MgATP concentration

is consistent with previous data reported for the rat liver

isoform [33,34] With MgATP as the variable substrate,

a hyperbolic curve was obtained at a high Glc

concen-tration (60 mm), with a Kmof 0.16 ± 0.01 mm (Table 3;

Fig 7C) However, at a low Glc concentration

(0.5 mm), negative cooperativity was observed with

respect to MgATP (nH= 0.87 ± 0.06) (Fig 7D),

con-sistent with a previous report on the rat liver isoform

[34], and the Km was reduced to 0.04 ± 0.003 mm

Interestingly, the L146R mutant, with a severely

reduced affinity for Glc (Table 1), demonstrated similar

negative kinetic cooperativity with respect to MgATP as

the variable substrate (nH= 0.73 ± 0.04)

Discussion

The bisubstrate reaction catalysed by monomeric GK

is mechanistically characterized by diffusion-controlled

binding of Glc to thermodynamically favoured

ligand-free conformations of the enzyme (Scheme 1), followed

by global hysteretic isomerization of the enzyme to a

closed conformation [29,31]

From crystallographic, biophysical and kinetic

stud-ies on GK, it is known that both substrate binding

and catalysis require substantial conformational

changes in the enzyme Ligand-free hGK is

structur-ally dominated by a super-open conformation [31],

which, in the crystal structure, is locked in an inactive

state by electrostatic and hydrophobic interactions

between the C-terminal helix (helix 17) and helix 6

[18] Three residues (Asn204, Asn231, and Glu256) in

the large domain [31] function as primary contact

res-idues in the binding of Glc [18,31] Pre-steady-state

analyses of Glc binding to WT hGK [26,32,35] have

provided evidence that the ligand-free enzyme in

solu-tion is in a pre-existing equilibrium between at least

two conformers (marked as GK and GK„ in

Scheme 1), i.e the super-open conformation ( 80–

95%) and an alternative (presumably less open)

con-formation ( 5–20%) with a higher affinity for Glc [26,35], which adds to the kinetic complexity of this reaction Recent high-resolution NMR analyses and pre-steady-state Glc binding experiments also suggest that GK is capable of sampling multiple conforma-tional states, both in the absence and the presence of Glc [32,36] The global conformational changes trig-gered by Glc binding have been defined crystallo-graphically [31] In the closed conformation (marked

as GK* in Scheme 1), precise alignment of additional substrate contact residues (notably Thr168 and Lys169 in the flexible surface⁄ active site loop) [18,29] and the subsequent higher affinity for Glc efficiently accelerate the chemical reaction (k3) on binding of the cosubstrate MgATP The overall binding constant

K1 for Glc and the values for the forward (k2) and reverse (k)2) rates of the conformational transition, which probably includes intermediates [29,31,35,36], have been estimated by stopped-flow fluorescence spectroscopy [25] In that study, the GK– Glc M GK*–Glc interconversion was found to

be slow, with k2= 0.45 s)1 and k)2= 0.28 s)1 (K2= 1.6), favouring the forward rate and isomeriza-tion, whereas the isomerization was unfavourable with 2-deoxyglucose as the substrate (K2= 0.8) Here, we present experimental evidence that ATP binds to the ligand-free form, and that this also results in changes

in the protein conformation

ATP binds to the ligand-free open conformation

of hGK Previous attempts to demonstrate direct binding

of ATP to the ligand-free form of rat GK by ITF

Table 3 The kinetic constants for WT GST–hGK at high and low concentrations of the fixed substrate The catalytic activity was measured

at 37 C, as described in Experimental procedures Kinetic parameters were calculated by nonlinear regression analyses with the Hill and Michaelis–Menten equations.

Glc as variable substrate

ATP as variable substrate

GK + Glc

GK‡ / ≠ ·Glc GK*·Glc GK*·Glc6P GK + Glc6P

GK≠ + Glc K1

ADP MgATP

Scheme 1 Reaction scheme for mammalian glucokinase.

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spectroscopy [37] and hGK by differential scanning

calorimetry [25] were reported to be unsuccessful The

topic was more recently readdressed [26] with a

non-hydrolysable ATP analogue (AMP-PNP) and ITF,

and relatively large quenching of the fluorescence

sig-nal was demonstrated, interpreted as a

nucleotide-induced conformational change However, as no

cor-rections were made for a large inner filter effect,

owing to the significant absorbance of the nucleotide

at the excitation wavelength (285 nm), we have

cor-rected for this effect here (at kex= 295 nm) as well

as for any effect of nonspecific binding to the enzyme

(i.e not in the active site), with the non-ATP-binding

mutant form T228M (Table 1) as a reference enzyme

Our analyses revealed that AMP-PNP and ATP do

indeed bind to hGK (Fig 2) in the ligand-free open

conformation, and the MD simulations (Fig 6)

fur-ther support this conclusion and also show the

resi-dues (including the mutated residue Thr228) directly

contacting ATP at the active site of WT hGK

The partial quenching effect of AMP-PNP on the

T228M reference enzyme with disrupted ATP binding

at the active site (Fig 2A,C) suggests a contribution

of nonspecific binding of that nucleotide (i.e not in

the active site) in addition to its inner filter effect,

as observed for free Trp The idea that AMP, in contrast to MgADP)⁄ MgATP2), can bind to more than one site has been suggested for the rat liver isoform [16]

Binding of ATP to ligand-free hGK results in a conformational change

High-resolution NMR analyses [36] have revealed that

GK is an intrinsically mobile enzyme whose structure and dynamics are modulated by temperature and ligand binding Here, we provide the first experimental evidence of ATP-dependent structural changes in WT hGK Specifically, our ITF quenching (Fig 2) and

MD simulations (Figs 5 and 6) indicate a significant conformational change upon ATP binding, including motion of the flexible surface⁄ active site loop and par-tial closure of the active site cleft (Figs 5C, 6B,C and S3) A change in conformation is further supported

by the significant protective effect of ATP on binding

of the extrinsic fluorescence probe ANS (Fig 3A,B) and on the limited proteolysis by trypsin (Fig 3C) In both assay systems, Glc showed more potent inhibi-tion than ATP, which may be related to the larger conformational change and more effective closure of

A

C

B

D

Fig 7 Steady-state kinetic properties of WT GST–hGK with Glc and MgATP as the variable substrates (A) At 5 m M MgATP, positive coo-perativity with respect to Glc was observed (n H = 1.95 ± 0.10) (B) At a low (0.05 m M ) concentration of MgATP, the cooperativity with respect to Glc was reduced (nH= 1.07 ± 0.07) (C) At 60 m M Glc, the binding curve for MgATP was hyperbolic (nH= 1.15 ± 0.04) (D) At a low (0.5 m M ) concentration of Glc, negative cooperativity with respect to MgATP binding was observed (nH= 0.87 ± 0.06) For all nonlinear regressions, the correlation coefficient (r 2 ) was > 0.99 The steady-state kinetic constants are summarized in Table 3.

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the active site cleft induced by Glc binding (Fig S3;

Table S2)

Kinetic cooperativity with respect to Glc

In general, the mechanism for cooperativity observed

in enzyme kinetic studies represents an experimental

challenge For monomeric GK, several models have

been considered to explain the positive kinetic

cooper-ativity with respect to Glc, including: (a) a random

order mechanism of substrate (Glc and MgATP2))

addition [13,38]; and (b) a sequential order mechanism

[15,39,40], in which the binding of Glc as the first

sub-strate induces a slow, concentration-dependent

confor-mational transition [34,40] characteristic of a hysteretic

enzyme [41,42] (Scheme 1) The Glc-induced

multipha-sic ITF enhancement (millisecond to minute time scale)

of WT and mutant forms of GK [18,25,26,35,37,43]

strongly favours the second mechanism, and support

the idea that the cooperativity can be explained by an

equilibrium between conformational states with

differ-ent affinities for Glc [18,25,26,35,37,43] However, little

experimental effort has been made to include or

exclude any contribution of (Mg)ATP binding to the

kinetic cooperativity

The ligand-free and the binary GK–Glc complex are

dynamic entities [32,36], and binding of (Mg)ATP may

shift the equilibrium between different enzyme

confor-mations, as shown for Glc [18,25,26,35,37,43] In this

study, the binding of ATP to the ligand-free enzyme

was found, by four independent criteria, to trigger

con-formational changes, including partial closure of the

active site cleft (Figs 5B, 6B,C and S3) Moreover,

pre-vious [34] and present (Table 1) steady-state kinetic

analyses are also compatible with conformational

con-trol of GK catalytic activity by the binding of

(Mg)ATP, with possible implications for kinetic

coo-perativity with respect to Glc Thus, the coocoo-perativity

is largely reduced (nH= 1.07 ± 0.07) at low

concen-trations of MgATP (Fig 7B; Table 1) [34] and when

MgATP is replaced by MgITP, a poor phosphoryl

donor ATP analogue [44]

In most previous steady-state kinetic analyses, rat GK

(liver) was observed to be noncooperative with respect

to (Mg)ATP [33,45,46] However, Neet et al [34]

reported negative kinetic cooperativity (nH= 0.84)

when measurements were made in the presence of 30%

glycerol at a low Glc concentration (0.5 mm), and this

was also observed here for the recombinant pancreatic

hGK in the absence of glycerol (nH= 0.87 ± 0.06)

However, when hGK activity was measured at high

glu-cose concentrations, the Hill coefficient for (Mg)ATP

approached 1.0 (Fig 7C), as expected from studies on

rat liver GK [33,45,46] Negative cooperativity (nH= 0.73 ± 0.04) was also observed for the GCK-MODY mutant L146R, which has severely reduced affinity for Glc (Table 1) Moreover, our studies on this mutant revealed that the analogue ATPcS (at 2.5 mm) increases the mutant’s low equilibrium binding affinity for Glc (Kd decreases from 19.3 ± 3.8 mm to 14.0 ± 1.4 mm), as well as the Glc-induced fluorescence enhancement (by 25%) (Fig 4) These effects may be related to partial catalytic activation following (Mg)ATP binding at physiological concentrations of Glc Similar or possibly larger effects of ATP in promot-ing a catalytically competent state may occur in other mutations associated with GCK-MODY

Conclusions

Using biochemical and biophysical methods, we have obtained experimental evidence in support of binding

of ATP to the ligand-free hGK, resulting in a change

of protein conformation The MD simulations indicate that the binding triggers molecular motion of the flexi-ble surface⁄ active site loop and partial closure of the interdomain active site cleft The modelled structure of the hGK–ATP binary complex shows the residue con-tacts involved in ATP binding at the active site Our findings further support conformational regulation of

GK by ATP binding, with possible implications for kinetic cooperativity with respect to Glc Further mutational studies, notably of GCK-MODY-associated mutations, may contribute to a better understanding

of the mechanistic and functional implications of the multiple conformational equilibria and the conforma-tional transitions induced by both Glc and (Mg)ATP, with possible future clinical implications

Experimental procedures

Materials

The oligonucleotide primers used for site-directed mutagen-esis were from Invitrogen (Carlsbad, CA, USA) The QuickChange XL Site-directed Mutagenesis Kit was from Stratagene (La Jolla, CA, USA) Glutathione Sepharose 4B was from Amersham Biosciences (GE Healthcare Europe GMBH, Oslo, Norway) Glc was from Calbiochem (San Diego, CA, USA) Magnesium chloride, magnesium ace-tate, guanidine hydrochloride, trypsin (bovine pancreas), trypsin inhibitor (soybean), pyruvate kinase (rabbit muscle), phospho(enol)pyruvate, ATP, ANS and AMP-PNP were from Sigma-Aldrich (St Louis, MO, USA) ATPcS was obtained from Roche Diagnostics Corporation (Indianapo-lis, IN, USA) All chemicals and buffers used for fluores-cence measurements were of the highest analytical grade

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